Barbiroli B, Medori R, Tritschler H J, Klopstock T, Seibel P, Reichmann H, Iotti S, Lodi R, Zaniol P
Cattedra di Biochimica Clinica, Istituto di Patologia Speciale Medica D. Campanacci, Universita' di Bologna, Italy.
J Neurol. 1995 Jul;242(7):472-7. doi: 10.1007/BF00873552.
A woman affected by chronic progressive external ophthalmoplegia and muscle mitochondrial DNA deletion was studied by phosphorus magnetic resonance spectroscopy (31P-MRS) prior to and after 1 and 7 months of treatment with oral lipoic acid. Before treatment a decreased phosphocreatine (PCr) content was found in the occipital lobes, accompanied by normal inorganic phosphate (Pi) level and cytosolic pH. Based on these findings, we found a high cytosolic adenosine diphosphate concentration [ADP] and high relative rate of energy metabolism together with a low phosphorylation potential. Muscle MRS showed an abnormal work-energy cost transfer function and a low rate of PCr recovery during the post-exercise period. All of these findings indicated a deficit of mitochondrial function in both brain and muscle. Treatment with 600 mg lipoic acid daily for 1 month resulted in a 55% increase of brain [PCr], 72% increase of phosphorylation potential, and a decrease of calculated [ADP] and rate of energy metabolism. After 7 months of treatment MRS data and mitochondrial function had improved further. Treatment with lipoate also led to a 64% increase in the initial slope of the work-energy cost transfer function in the working calf muscle and worsened the rate of PCr resynthesis during recovery. The patient reported subjective improvement of general conditions and muscle performance after therapy. Our results indicate that treatment with lipoate caused a relevant increase in levels of energy available in brain and skeletal muscle during exercise.
一名患有慢性进行性眼外肌麻痹和肌肉线粒体DNA缺失的女性,在口服硫辛酸治疗前、治疗1个月后和7个月后接受了磷磁共振波谱(31P-MRS)研究。治疗前,枕叶中磷酸肌酸(PCr)含量降低,同时无机磷酸盐(Pi)水平和胞质pH值正常。基于这些发现,我们发现胞质二磷酸腺苷浓度[ADP]较高,能量代谢相对速率较高,而磷酸化电位较低。肌肉MRS显示运动后时期工作能量消耗转移功能异常,PCr恢复率较低。所有这些发现均表明脑和肌肉中线粒体功能存在缺陷。每天服用600毫克硫辛酸治疗1个月后,脑[PCr]增加了55%,磷酸化电位增加了72%,计算出的[ADP]和能量代谢速率降低。治疗7个月后,MRS数据和线粒体功能进一步改善。用硫辛酸治疗还导致工作中的小腿肌肉工作能量消耗转移功能的初始斜率增加了64%,并使恢复期间PCr再合成速率恶化。患者报告治疗后总体状况和肌肉性能有主观改善。我们的结果表明,硫辛酸治疗导致运动期间脑和骨骼肌中可用能量水平有显著增加。